Introduction: Cyclopentolate is a widely used mydriatic agent with a rapid onset cycloplegic effect. Systemic toxicity from an ophthalmic application is uncommon. Case Presentation: We present a case of a 13-year-old girl who presented to the emergency department with a generalized seizure after receiving 1% cyclopentolate for three drops in each eye, over 15 minutes, as instructed by the prescriber. The seizure began 30 minutes after receiving the last dose. We also describe the general presentation, characteristics of affected patients, and management of this adverse drug event. Cyclopentolate systemic toxicity is dose-dependent. Conclusion: This case highlights the importance of caution when using topical ophthalmic cycloplegics in children.

Keywords: Cyclopentolate 1%, eye drops, cycloplegic, seizure, case report, ophthalmic eye drops, case report.